Crawley article on What Matters To Children With cfs/ME

"What matters to children with CFS/ME? A conceptual model as the first stage in developing a PROMAbstract

Background Paediatric chronic fatigue syndrome (CFS)/myalgic encephalomyelitis (ME) is relatively common and disabling. Research is hampered because current patient-reported outcome measures (PROMs) do not capture outcomes that are important to children with CFS/ME.

Aim The aim of this study was to explore the aspects of life and health outcomes that matter to children with CFS/ME.

Methods Twenty-five children with CFS/ME were interviewed (11 males, 14 females; mean age 12.9 years (SD 2.2), range 8–17). Twelve were trial participants interviewed during the trial and 13 were recruited as part of a follow-up qualitative study. Parents were present in 19 interviews with their children. Three mothers participated in a focus group. All the interviews and the focus group were audio-recorded and transcribed. Data were analysed thematically using techniques of constant comparison. NVivo was used to structure and categorise data in a systematic way.

Results Children identified four key themes (health outcome domains): ‘symptoms’ that fluctuated, which caused an unpredictable reduction in both ‘physical activity’ and ‘social participation’ all of which impacted on ‘emotional well-being’. These domains were influenced by both ‘management’ and ‘contextual factors’, which could be positive and negative. The relationship between healthcare and school was considered pivotal.

Conclusions Children's descriptions helped to inform a conceptual model that is necessary to develop a new paediatric CFS/ME PROM. Doctors need to be aware of how children conceptualise CFS/ME; the relationship between healthcare and school is fundamental to ameliorate the impact of CFS/ME.

Kirstie Hayward is keen to involve patients (and oddly thinks that patients should be consulted on what outcomes matters to them), and is running a workshop for patients and researchers and the forthcoming CMRC conference this week which might ultimately lead to a new mecfs PROM for adults:

Current PROMs used for CFS/ME measure fatigue, disability and mood. A new PROM needs to collect data on symptoms, activity and socialising. Most of these themes are not currently assessed by PROMs used in paediatric CFS/ME.18 Future research should explore which outcomes are most important to children to include in a new PROM.

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So this might have been done as an excuse to move (even further) away from potentially objective outcome measurements.

So this might have been done as an excuse to move (even further) away from potentially objective outcome measurements.

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Not really. By definition, a PROM is self-reported, and ideally focusing on what patients say matters most to them. That's separate from objective measures.

The generic problem with all self-reports is that they are prone to bias, and while this tends to be a smallish effect it can be a big issue in unblinded trials with no proper control groups where results are only modest (insert your own favourite example here). Personally, while I'm a fan of good PROMs, I think objective measures should always be used alongside them (and iniital validation of PROMs should require showing that any 'change' shown in clinical trials is matched by objective measures).

As Jonathan Edwards has said elsewhere, a treatment (in any illness) that improves (objective) biological markers but doesn't leave patients feeling any better isn't going to impress many patients. We need both measures.

That was my first thought too! But I was surprised: I think they've actually done a good/honest job of listening here. Perhaps, as Simon points out, because Crawley's not the most important author here. And I think their outcomes/conclusions are quite good too. I'm quite impressed by it actually.

That was my first thought too! But I was surprised: I think they've actually done a good/honest job of listening here. Perhaps, as Simon points out, because Crawley's not the most important author here. And I think their outcomes/conclusions are quite good too. I'm quite impressed by it actually.

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She's the corresponding author, which is usually the most important one

It's just hard to believe that anything good could come from Crawley, and without the ultimate intention of it being warped into something abusive.

Over the years I've read occasional anecdotes about Crawley being too willing to diagnose "pervasive refusal syndrome" in children or teenagers who fail to recover as quickly as expected or who are non-compliant with graded increases in activity. These patients are then regarded as "recovered" from ME/CFS based on the notion that the illness has resolved as expected but now the patients are recalcitrant or avoidant. Anyone know more about that, are there any publicly known cases? I imagine that disputable alternative diagnoses would matter to children with ME/CFS too.

She's the corresponding author, which is usually the most important one

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I meant important in terms of the amount of input into the study. It doesn't have Crawley's finger prints all over the text of the study so I'm assuming she was fairly hands off, and that others took the lead. Simon's comments confirmed my thoughts about that. I might be wrong about that, of course, but I'm still impressed with the study.

As usual, these studies (SMILE and PROMs) only dealt with mild to moderately affected young people, defined by NICE guidelines and I won't go into those limitations.

There is no talk of PEM or OI ( in my opinion - dizziness and sweats aren't accurate characterizations of OI, if that is what the authors intended) or how they impact the young people.

No fathers were interviewed.

"Supportive schools, families and friends and strategies to manage activity can help ameliorate symptoms."
I don't think they ameliorate symptoms but they do lessen the constant disbelief patients encounter.

PROMs "Participants were also less anxious and less likely to be female because we prospectively recruited male patients..." Is it just me or does this sound as though they imply that female patients are more anxious than male?

"Supportive schools, families and friends and strategies to manage activity can help ameliorate symptoms."
I don't think they ameliorate symptoms but they do lessen the constant disbelief patients encounter.

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If you have a supportive school that allows e.g. home schooling then that can help manage activity levels which helps manage symptoms. A supportive family can definitely help manage activity levels too. But their phrasing is unhelpful.

"Participants were also less anxious and less likely to be female because we prospectively recruited male patients..." Is it just me or does this sound as though they imply that female patients are more anxious than male?

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I didn't understand that either. I think it might just be bad phrasing and they might just mean "participants were less anxious" and also "less likely to be female because we prospectively recruited male patients".

There is no talk of PEM or OI ( in my opinion - dizziness and sweats aren't accurate characterizations of OI, if that is what the authors intended) or how they impact the young people.

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Yep, post exertional malaise doesn't seem to feature in their vocabulary, which is quite an omission. Orthostatic intolerance is not often featured in CFS research, so I'm not surprised that they don't discuss that. Both omissions suggest that the authors have a lot to learn.

The generic problem with all self-reports is that they are prone to bias, and while this tends to be a smallish effect

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Generally I think this is right. However I strongly suspect that when the primary intervention is psychotherapy designed to change patient thinking the bias in self reporting is most likely the dominant effect. Even if the patients get worse the subjective outcome might seem like a raging success.